Evidence map›Paper›PMID 40128563›Full record

ReviewLeukemia2025

Overt and covert genetic causes of pediatric acute lymphoblastic leukemia.

Ulrik Stoltze, Stefanie V Junk, Anna Byrjalsen, Hélène Cavé, Giovanni Cazzaniga, Sarah Elitzur, Eva Fronkova, Lisa Lyngsie Hjalgrim, Roland P Kuiper, Louise Lundgren and 9 more

Abstract readReview
PubMed Publisher
In one paragraph

Review in Leukemia, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

6 citing papers in PubMed.

  1. Article
  2. Review
  3. Review
  4. Article
  5. Article
  6. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

19 authors.

Ulrik StoltzeDepartment of Childhood and Adolescent Medicine, Rigshospitalet, Copenhagen, Denmark. ulrik.kristoffer.stoltze@regionh.dk.ORCID 0000-0001-5862-3292
Stefanie V JunkDepartment of Pediatric Oncology, Hematology and Clinical Immunology, Medical Faculty, Heinrich Heine University Düsseldorf, Düsseldorf, Germany.ORCID 0000-0001-8206-9463
Anna ByrjalsenDepartment of Childhood and Adolescent Medicine, Rigshospitalet, Copenhagen, Denmark.
Hélène CavéDepartment of Genetics, Robert Debré University Hospital, APHP, Paris, France.ORCID 0000-0003-2840-1511
Giovanni CazzanigaTettamanti Center, Fondazione IRCCS San Gerardo dei Tintori, Monza, Italy.
Sarah ElitzurDepartment of Pediatric Hematology and Oncology, Schneider Children's Medical Center and Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.ORCID 0000-0002-3495-7578
Eva FronkovaChildhood Leukaemia Investigation Prague, Department of Paediatric Haematology and Oncology, Second Faculty of Medicine, Charles University and University Hospital Motol, Prague, Czechia.ORCID 0000-0002-6900-8145
Lisa Lyngsie HjalgrimDepartment of Childhood and Adolescent Medicine, Rigshospitalet, Copenhagen, Denmark.
Roland P KuiperPrincess Máxima Center for Pediatric Oncology, Utrecht, Netherlands.ORCID 0000-0003-4928-3809
Louise LundgrenDepartment of Childhood and Adolescent Medicine, Rigshospitalet, Copenhagen, Denmark.
Melina MescherDepartment of Pediatric Oncology, Hematology and Clinical Immunology, Medical Faculty, Heinrich Heine University Düsseldorf, Düsseldorf, Germany.
Theis MikkelsenDepartment of Childhood and Adolescent Medicine, Rigshospitalet, Copenhagen, Denmark.ORCID 0000-0003-2748-4001
Agata PastorczakDepartment of Pediatrics, Oncology, and Hematology, Medical University of Lodz, Lodz, Poland.
Marion StrulluUniversity Paris Cité, Paris, France.
Jan TrkaChildhood Leukaemia Investigation Prague, Department of Paediatric Haematology and Oncology, Second Faculty of Medicine, Charles University and University Hospital Motol, Prague, Czechia.ORCID 0000-0002-9527-8608
Karin WadtDepartment of Clinical Genetics, Rigshospitalet, Copenhagen, Denmark.
Shai IzraeliDepartment of Pediatric Hematology and Oncology, Schneider Children's Medical Center and Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.ORCID 0000-0002-6938-2540
Arndt BorkhardtDepartment of Pediatric Oncology, Hematology and Clinical Immunology, Medical Faculty, Heinrich Heine University Düsseldorf, Düsseldorf, Germany.ORCID 0000-0002-6121-4737
Kjeld SchmiegelowDepartment of Childhood and Adolescent Medicine, Rigshospitalet, Copenhagen, Denmark. kjeld.schmiegelow@regionh.dk.ORCID 0000-0002-0829-4993

Funding

Associazione Italiana per la Ricerca sul Cancro (Italian Association for Cancer Research) 2023-29175Børnecancerfonden (Danish Childhood Cancer Foundation) 2020-5769Børnecancerfonden (Danish Childhood Cancer Foundation) 2023-001063Kræftens Bekæmpelse (Danish Cancer Society) R-257-A14720
6 · The paper itself

Abstract

Pediatric acute lymphoblastic leukemia (pALL) is the most common childhood malignancy, yet its etiology remains incompletely understood. However, over the course of three waves of germline genetic research, several non-environmental causes have been identified. Beginning with trisomy 21, seven overt cancer predisposition syndromes (CPSs)-characterized by broad clinical phenotypes that include an elevated risk of pALL-were first described. More recently, newly described CPSs conferring high risk of pALL are increasingly covert, with six exhibiting only minimal or no non-cancer features. These 13 CPSs now represent the principal known hereditary causes of pALL, and human pangenomic data indicates a strong negative selection against mutations in the genes associated with these conditions. Collectively they affect approximately 1 in 450 newborns, of which just a minority will develop the disease. As evidenced by tailored leukemia care protocols for children with trisomy 21, there is growing recognition that CPSs warrant specialized diagnostic, therapeutic, and long-term management strategies. In this review, we investigate the evidence that the 12 other CPSs associated with high risk of pALL may also see benefits from specialized care - even if these needs are often incompletely mapped or addressed in the clinic. Given the rarity of each syndrome, collaborative international research and shared data initiatives will be crucial for advancing knowledge and improving outcomes for these patients.

Indexed as

Genetic Predisposition to DiseasePrecursor Cell Lymphoblastic Leukemia-LymphomaChildHumans

Identifiers

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.